Literature DB >> 2783588

Identification of a point mutation resulting in a heat-labile adenosine deaminase (ADA) in two unrelated children with partial ADA deficiency.

R Hirschhorn1, S Tzall, A Ellenbogen, S H Orkin.   

Abstract

We have determined the mutation in a child with partial adenosine deaminase (ADA) deficiency who is phenotypically homozygous for a mutant ADA gene encoding a heat-labile enzyme (Am. J. Hum. Genet. 38: 13-25). Sequencing of cDNA demonstrated a C to A transversion that results in the replacement of a proline by a glutamine residue at codon 297. As this mutation generated a new recognition site in exon 10 of genomic DNA for the enzyme Alu I, Southern blot analysis was used to establish that this child was indeed homozygous for the mutation. The abnormal restriction fragment generated by this mutation was also found in a second partially ADA-deficient patient who phenotypically is a genetic compound and also expresses a heat-labile ADA (in addition to a more acidic than normal ADA) (Am. J. Hum. Genet. 38: 13-25). Sequencing of cDNA clones from the second patient established the identical codon 297 mutation. Transfection of the mutant cDNA into heterologous cells resulted in expression of a heat-labile ADA of normal electrophoretic mobility and isoelectric point, properties exhibited by the ADA in the patients' cells.

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Year:  1989        PMID: 2783588      PMCID: PMC303706          DOI: 10.1172/JCI113909

Source DB:  PubMed          Journal:  J Clin Invest        ISSN: 0021-9738            Impact factor:   14.808


  27 in total

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2.  Complete sequence and structure of the gene for human adenosine deaminase.

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5.  Analysis of the accuracy and implications of simple methods for predicting the secondary structure of globular proteins.

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6.  Severe combined immune deficiency due to a homozygous 3.2-kb deletion spanning the promoter and first exon of the adenosine deaminase gene.

Authors:  T M Berkvens; E J Gerritsen; M Oldenburg; C Breukel; J T Wijnen; H van Ormondt; J M Vossen; A J van der Eb; P Meera Khan
Journal:  Nucleic Acids Res       Date:  1987-11-25       Impact factor: 16.971

7.  Mutations in the human adenosine deaminase gene that affect protein structure and RNA splicing.

Authors:  A L Akeson; D A Wiginton; J C States; C M Perme; M R Dusing; J J Hutton
Journal:  Proc Natl Acad Sci U S A       Date:  1987-08       Impact factor: 11.205

8.  Isolation of a cDNA for human acid alpha-glucosidase and detection of genetic heterogeneity for mRNA in three alpha-glucosidase-deficient patients.

Authors:  F Martiniuk; M Mehler; A Pellicer; S Tzall; G La Badie; C Hobart; A Ellenbogen; R Hirschhorn
Journal:  Proc Natl Acad Sci U S A       Date:  1986-12       Impact factor: 11.205

9.  DNA sequencing with chain-terminating inhibitors.

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10.  Molecular cloning of the murine adenosine deaminase gene from a genetically enriched source: identification and characterization of the promoter region.

Authors:  D E Ingolia; M R Al-Ubaidi; C Y Yeung; H A Bigo; D Wright; R E Kellems
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  6 in total

1.  Identification and characterization of nine RFLPs at the adenosine deaminase (ADA) locus.

Authors:  S Tzall; A Ellenbogen; F Eng; R Hirschhorn
Journal:  Am J Hum Genet       Date:  1989-06       Impact factor: 11.025

2.  Homozygosity for a newly identified missense mutation in a patient with very severe combined immunodeficiency due to adenosine deaminase deficiency (ADA-SCID).

Authors:  R Hirschhorn; V Chakravarti; J Puck; S D Douglas
Journal:  Am J Hum Genet       Date:  1991-10       Impact factor: 11.025

3.  Hot spot mutations in adenosine deaminase deficiency.

Authors:  R Hirschhorn; S Tzall; A Ellenbogen
Journal:  Proc Natl Acad Sci U S A       Date:  1990-08       Impact factor: 11.205

4.  Somatic mosaicism for a newly identified splice-site mutation in a patient with adenosine deaminase-deficient immunodeficiency and spontaneous clinical recovery.

Authors:  R Hirschhorn; D R Yang; A Israni; M L Huie; D R Ownby
Journal:  Am J Hum Genet       Date:  1994-07       Impact factor: 11.025

5.  Paradoxical expression of adenosine deaminase in T cells cultured from a patient with adenosine deaminase deficiency and combine immunodeficiency.

Authors:  F X Arredondo-Vega; J Kurtzberg; S Chaffee; I Santisteban; E Reisner; M S Povey; M S Hershfield
Journal:  J Clin Invest       Date:  1990-08       Impact factor: 14.808

6.  RNA editing of BFP, a point mutant of GFP, using artificial APOBEC1 deaminase to restore the genetic code.

Authors:  Sonali Bhakta; Matomo Sakari; Toshifumi Tsukahara
Journal:  Sci Rep       Date:  2020-10-14       Impact factor: 4.379

  6 in total

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